ArticleThe Journal of neuroscience : the official journal of the Society for Neuroscience2016
Neuroplastin Isoform Np55 Is Expressed in the Stereocilia of Outer Hair Cells and Required for Normal Outer Hair Cell Function.
Article in The Journal of neuroscience : the official journal of the Society for Neuroscience, 2016. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 22 papers.
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Who cites it
22 citing papers in PubMed, 29 citations in OpenAlex.
- CaProceedings of the National Academy of Sciences of the United States of America · 2026Article
- Deafness causing neuroplastin missense variants fail to promote plasma membrane CaThe Journal of biological chemistry · 2024Article
- Neuroplastin Expression in Male Mice Is Essential for Fertility, Mating, and Adult Testosterone Levels.International journal of molecular sciences · 2023Article
- Absence of Embigin accelerates hearing loss and causes sub-viability, brain and heart defects in C57BL/6N mice due to interaction withiScience · 2023Article
- Linear mixed-effect modeling of organ of Corti vibratory tuning curves.Hearing research · 2023Article
- Review
- MANF supports the inner hair cell synapse and the outer hair cell stereocilia bundle in the cochlea.Life science alliance · 2022Article
- Neuroplastin genetically interacts with Cadherin 23 and the encoded isoform Np55 is sufficient for cochlear hair cell function and hearing.PLoS genetics · 2022Article
- Neuroplastin in Neuropsychiatric Diseases.Genes · 2021Review
- Neuroplastin expression is essential for hearing and hair cell PMCA expression.Brain structure & function · 2021Article
- Retrograde Amnesia - A Question of Disturbed Calcium Levels?Frontiers in cellular neuroscience · 2021Review
- Serial scanning electron microscopy of anti-PKHD1L1 immuno-gold labeled mouse hair cell stereocilia bundles.Scientific data · 2020Article
- The Interaction of TRAF6 With Neuroplastin Promotes Spinogenesis During Early Neuronal Development.Frontiers in cell and developmental biology · 2020Article
- Hair-Bundle Links: Genetics as the Gateway to Function.Cold Spring Harbor perspectives in medicine · 2019Review
- PKHD1L1 is a coat protein of hair-cell stereocilia and is required for normal hearing.Nature communications · 2019Article
- Molecular Composition of Vestibular Hair Bundles.Cold Spring Harbor perspectives in medicine · 2019Review
- Microarray Analysis of Gene Expression Changes in Neuroplastin 65-Knockout Mice: Implications for Abnormal Cognition and Emotional Disorders.Neuroscience bulletin · 2018Article
- A Model for Link Pruning to Establish Correctly Polarized and Oriented Tip Links in Hair Bundles.Biophysical journal · 2017Article
- A large scale hearing loss screen reveals an extensive unexplored genetic landscape for auditory dysfunction.Nature communications · 2017Article
- A complex of Neuroplastin and Plasma Membrane CaScientific reports · 2017Article
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Authors and funding
8 authors at 5 institutions in 1 country.
Funding
Abstract
unlabelledNeuroplastin (Nptn) is a member of the Ig superfamily and is expressed in two isoforms, Np55 and Np65. Np65 regulates synaptic transmission but the function of Np55 is unknown. In an N-ethyl-N-nitrosaurea mutagenesis screen, we have now generated a mouse line with an Nptn mutation that causes deafness. We show that Np55 is expressed in stereocilia of outer hair cells (OHCs) but not inner hair cells and affects interactions of stereocilia with the tectorial membrane. In vivo vibrometry demonstrates that cochlear amplification is absent in Nptn mutant mice, which is consistent with the failure of OHC stereocilia to maintain stable interactions with the tectorial membrane. Hair bundles show morphological defects as the mutant mice age and while mechanotransduction currents can be evoked in early postnatal hair cells, cochlea microphonics recordings indicate that mechanontransduction is affected as the mutant mice age. We thus conclude that differential splicing leads to functional diversification of Nptn, where Np55 is essential for OHC function, while Np65 is implicated in the regulation of synaptic function. SIGNIFICANCE STATEMENT: Amplification of input sound signals, which is needed for the auditory sense organ to detect sounds over a wide intensity range, depends on mechanical coupling of outer hair cells to the tectorial membrane. The current study shows that neuroplastin, a member of the Ig superfamily, which has previously been linked to the regulation of synaptic plasticity, is critical to maintain a stable mechanical link of outer hair cells with the tectorial membrane. In vivo recordings demonstrate that neuroplastin is essential for sound amplification and that mutation in neuroplastin leads to auditory impairment in mice.
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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.